Year 2022 / Volume 29 / Number 1

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Cilinical Note

Bone pain management with opioid medication in a patient with Camurati-Engelmann disease: a case report

Rev. Soc. Esp. Dolor. 2022; 29(1): 51-55 / DOI: 10.20986/resed.2021.3930/2021

Leonardo Arce, María Paz Grisales, Katalina Espinosa, Catalina Baena


ABSTRACT

Introduction: Camurati-Engelman Disease is a rare genetic sclerosing bone dysplasia with periosteal and endosteal thickening of the cortical of the long bones. It generates pain secondary to the reduction of the medullary canal that is usually controlled with corticosteroids and, in severe cases, with surgical decompression.
Case history: We present the case of a woman with a genetic diagnosis of Camurati-Engelman Disease with poor pain control with corticosteroid management and surgical procedures throughout her childhood and early adulthood. In whom optimal pain control was achieved with pain regimen with hydrocodone analgesic management. This is the first case described in the literature for adequate pain control using an opioid drug.
Discussion: CE disease is an extremely rare genetic entity with little more than 300 cases reported in the world. It is generated by an alteration in the gene for growth factor-beta 1 (TGF-B1); it has a varied clinical presentation that can begin with bone alterations accompanied by muscle weakness, joint angular alterations, headache, and nerve compressions. It has a differential diagnosis with some genetic entities that may present clinical similarity, but its morphological and radiological characteristics are distinctive. The usual management of bone pain generated by this entity is based on corticosteroids, in addition to losartan or surgical intervention aimed at reducing cortical changes. The intervention with opioid analgesics accompanied by a rehabilitation plan is not a frequent report, this being a case of success due to the refractoriness of the symptoms in a patient with chronic pain, with a positive impact on her functionality and quality of life.
Conclusion: It is considered that analgesic management with opioids may be a treatment option in patients with Camurati-Engelman disease refractory to corticosteroid management and surgical interventions.



RESUMEN

Introducción: La enfermedad de Camurati-Engelman (CE) es una displasia ósea esclerosante rara, de causa genética. Se presenta con engrosamiento perióstico y endóstico de la cortical de los huesos largos. Genera dolor secundario a la reducción del canal medular, que habitualmente se controla con corticoides y en casos severos, con descompresión quirúrgica.
Historia del caso: Presentamos el caso de una mujer con diagnóstico genético de enfermedad de Camurati-Engelman, con mal control del dolor, con manejo de corticosteroides y procedimientos quirúrgicos a lo largo de su niñez y adultez temprana. Se logró un control óptimo del dolor con un régimen con manejo analgésico con hidrocodona. Este es el primer caso descrito en la literatura de un adecuado control del dolor con un medicamento opioide.
Discusión: La enfermedad de CE es una entidad genética extremadamente rara, con poco más de 300 casos reportados en el mundo. Se genera por una alteración en el gen del factor de crecimiento beta 1 (TGF-B1). Tiene una presentación clínica variada que puede iniciar con las alteraciones óseas acompañado de debilidad muscular, alteraciones angulares articulares, cefalea y compresiones nerviosas. Tiene diagnóstico diferencial con algunas entidades genéticas que pueden presentar similitud clínica, pero su característica morfológica y radiológica es distintiva. El manejo usual del dolor óseo generado por esta entidad se basa en corticoesteroides, además de losartán o intervenciones quirúrgicas orientadas a disminuir los cambios corticales. La intervención con analgésicos opioides, acompañada de un plan de rehabilitación, no es un reporte frecuente, siendo este un caso de éxito ante la refractariedad de los síntomas en una paciente con dolor crónico, impactando de manera positiva en su funcionalidad y calidad de vida.
Conclusión: Se considera que el manejo analgésico con opioides puede ser una opción de tratamiento en pacientes con enfermedad de Camurati-Engelman refractaria al manejo con corticoides e intervenciones quirúrgicas.





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Bibliografía

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Instrucciones para citar

Arce L, Grisales M, Espinosa K, Baena C. Bone pain management with opioid medication in a patient with Camurati-Engelmann disease: a case report. Rev Soc Esp Dolor 2022; 29(1): 51-55 / DOI: 1020986/resed20213930/2021


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Recibido: 01/06/2021

Aceptado: 20/11/2021

Prepublicado: 03/12/2021

Publicado: 05/08/2022

Tiempo de revisión del artículo: 63 días

Tiempo de prepublicación: 185 días

Tiempo de edición del artículo: 430 días


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